Retractions, corrections and expressions of concern in chronic fatigue syndrome research

From MEpedia, a crowd-sourced encyclopedia of ME and CFS science and history

Some research and scientific publications about chronic fatigue syndrome or myalgic encephalomyelitis have been withdrawn or corrected due to a range of different scientific issues.

Reasons for retractions and corrections[edit | edit source]

In ME/CFS research reasons have included:

  • scientific criticism of methodology
  • incorrect or missing mentions of the fact that primary outcomes had been swapped for pre-registered trials, particularly when objective primary outcomes were swapped for questionnaire results in non-blinded trials.
  • the merging of feasibility study data with trial data giving incorrect timeliness, and potentially biased results
  • scientific challenge of the conclusions, for example interpretation of the data
  • ethics approval concerns or incorrect ethics statements
  • lack of compliance to journal policies (e.g., data sharing or pre-registration of clinical trials)
  • lack of adherence to scientific standards
  • incorrect use of scientific assessment tools, e.g., reviews incorrectly using the GRADE framework to assess the risk of bias or other factors
  • also significant has been the inclusion or exclusion of patients using the Oxford criteria, or the exclusion of patients with severe and very severe ME, which affect whether results can be considered representative of all patients or may reflect patients with chronic fatigue not caused by chronic fatigue syndrome.

Other publications have faced widespread calls for retraction or independent re-analysis of the data, but have not been retracted, for example the main PACE trial publication.

Patient petitions, and pressure from patient groups without scientific criticism have not led to the cancellation of planned research, or to retractions or corrections, although some patient groups have signed open letters of scientific criticism by scientists or published their own scientific criticism.

Retracted articles[edit | edit source]

XMRV virus publications[edit | edit source]

Two publications reporting the presence of XMRV viral matter in patients with chronic fatigue syndrome were retracted after the studies failed replication, and the XMRV was found to be a laboratory contaminant.[citation needed] An earlier study on XMRV and prostate cancer was also retracted.

Withdrawn publications[edit | edit source]

Exercise therapy for chronic fatigue syndrome (individual patient data)[edit | edit source]

Published by Cochrane in 2014[1] the patient data for exercise therapy for chronic fatigue syndrome was withdrawn in 2018, the reason given was that the protocol was no longer being progressed to a review.[2]

Cochrane exercise therapy review[edit | edit source]

Cochrane announced their intention to withdraw the review of the use of Exercise therapy in chronic fatigue syndrome but later reversed this decision and made multiple amendments.[citation needed] The initial announcement was made after a detailed complaint was made demonstrating the failure of the review to confirm to Cochrane's published standards.

Editorial and author corrections[edit | edit source]

Cochrane exercise therapy review[edit | edit source]

The highly influential Cochrane review of exercise therapy for ME/CFS has had repeated corrections and editorial notes.

Amended ethics statements[edit | edit source]

Professor Esther Crawley, a British pediatrician, published eleven different research publications involving children which used an ethics approval number intended for a different purpose. These publications were later corrected to state that there was no ethics approval reference, and that they had been determined to be "service evaluations" which were exempt from requiring approval - however, some researchers and scientists disputed the service evaluation judgement.[3]

SMILE trial[edit | edit source]

This trial of a therapy called the Lightning Process was highly controversial, partly because it was a trial involving children as young as eight using a therapy never assessed in adults, which gave safety concerns. This trial, known as the SMILE trial, was republished with corrections after open letter of complaint including scientific criticism and calling for retraction.

The final version includes acknowledgement from the authors that the study was not fully ICMJE compliant. The process has additionally involved seeking assurance from the authors that the change in primary outcome was not influenced by (positive) findings in the feasibility phase... BMJ policy requires prospective registration of randomised trials but we do not consider a failure to enforce that policy grounds for retraction. - Nick Brown, Editor of Archives of Disease in Childhood (2019)
Extracts from the editorial correction of the SMILE trial for ME/CFS by Esther Crawley et al (2018).

An open letter was sent to the editor of Archives of Disease in Childhood in January 2018 by Dr David Tuller and over twenty other signatories for a correction to the serious issues and anomalies in the published paper.[5]

In July 2019, after an investigation by the Archives of Disease in Childhood, and significant correspondence between the study's authors, journal editor Nick Brown, and Dr David Tuller, a lengthy and detailed editorial correction to the SMILE trial was published but the journal refusal to retract the SMILE publication.[4]

Expressions of concern[edit | edit source]

PACE trial research[edit | edit source]

A PLOS ONE PACE trial study[6][7] on cost effectiveness of treatment. The Editors of PLOS One, Iratxe Puebla and Joerg Heber, wrote in PLOS One Blog "Since we feel we have exhausted the options to make the data available responsibly, and considering the questions that were raised about the validity of the article’s conclusions, we have decided to post an Expression of Concern to alert readers that the data are not available in line with the journal’s editorial policy".[8]

Calls for retraction or independent re-analysis of results[edit | edit source]

  • PACE trial main outcome publication - 3 open letters, re-analysis was later made possible by the release of the patient data ordered by a Freedom of Information Act tribunal in the UK
  • Cochrane exercise therapy review
  • A PACE trial publication by PLOS ONE
  • Journal of Health Psychology's special issue on the PACE trial, prior to publication an attempt was made to prevent publication
  • SMILE trial - editorial correction issued instead

See also[edit | edit source]

Learn more[edit | edit source]

References[edit | edit source]

  1. Larun, Lillebeth; Odgaard-Jensen, Jan; Brurberg, Kjetil G; Chalder, Trudie; Dybwad, Marianne; Moss-Morris, Rona E; Sharpe, Michael; Wallman, Karen; Wearden, Alison (April 1, 2014). "Exercise therapy for chronic fatigue syndrome (individual patient data)". Cochrane Database of Systematic Reviews. doi:10.1002/14651858.cd011040. ISSN 1465-1858. PMC 6494520.
  2. Larun, Lillebeth; Odgaard‐Jensen, Jan; Brurberg, Kjetil G.; Chalder, Trudie; Dybwad, Marianne; Moss‐Morris, Rona E.; Sharpe, Michael; Wallman, Karen; Wearden, Alison (2018). "Exercise therapy for chronic fatigue syndrome (individual patient data)". Cochrane Database of Systematic Reviews (12). doi:10.1002/14651858.CD011040.pub2. ISSN 1465-1858.
  3. "Correction: Obesity in adolescents with chronic fatigue syndrome: an observational study". Archives of Disease in Childhood. 105 (2): e1. February 2020. doi:10.1136/archdischild-2016-311293corr1. ISSN 1468-2044. PMC 7025721. PMID 31969354.
  4. 4.0 4.1 Brown, Nick (July 2019). "Editor's Note on Correction to Crawley et al 2018". Archives of Disease in Childhood.
  5. Tuller, David (January 30, 2018). "Trial By Error: A Letter to Archives of Disease in Childhood". Virology blog. Retrieved July 16, 2019.
  6. McCrone, Paul; Sharpe, Michael; Chalder, Trudie; Knapp, Martin; Johnson, Anthony L.; Goldsmith, Kimberley A.; White, Peter D. (August 1, 2012). "Adaptive Pacing, Cognitive Behaviour Therapy, Graded Exercise, and Specialist Medical Care for Chronic Fatigue Syndrome: A Cost-Effectiveness Analysis". PLOS ONE. 7 (8): e40808. doi:10.1371/journal.pone.0040808. ISSN 1932-6203. PMC 3411573. PMID 22870204.
  7. Puebla, Iratxe; Heber, Joerg (May 2, 2017). "Expression of Concern: Adaptive Pacing, Cognitive Behaviour Therapy, Graded Exercise, and Specialist Medical Care for Chronic Fatigue Syndrome: A Cost-Effectiveness Analysis". PLOS ONE. 12 (5): e0177037. doi:10.1371/journal.pone.0177037. ISSN 1932-6203. PMC 5412692. PMID 28463341.
  8. "Data sharing in clinical research: challenges and open opportunities". PLoS. May 2, 2017. Retrieved July 19, 2020.